Ectopic Parathyroid Adenoma in an 11-Year-Old Girl: Case Report and Literature Review

Flokas, Myrto Eleni; Ganieva, Gulnigor; Grieco, Amanda; Agdere, Levon

doi:10.1016/j.aace.2020.11.013

Cited by 6 publications

(14 citation statements)

References 36 publications

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“…The patient’s ectopic mediastinal parathyroid adenoma was resected using a thoracoscopic approach. This has been a common approach in adult patients for decades [ 13 ], but has been reported in just a handful of recent pediatric cases [ 14 , 15 ]. Preoperative localization studies and cross-sectional imaging are essential for operative planning.…”

Section: Discussionmentioning

confidence: 99%

“…To our knowledge, this is the first reported case of PHPT secondary to ectopic parathyroid adenoma presenting with bilateral SCFE. The thoracoscopic approach to ectopic parathyroid adenoma resection has also been reported only a few times in the literature [ 14 , 15 ]. This patient’s presentation underscores the severity with which PHPT can present in pediatric patients and the rapidity with which the hypercalcemia can progress.…”

Section: Discussionmentioning

confidence: 99%

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Primary Hyperparathyroidism From Ectopic Parathyroid Adenoma in a 12-Year-Old With Slipped Capital Femoral Epiphysis

Vitale

Shieh

Modi

et al. 2022

Journal of the Endocrine Society

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Primary hyperparathyroidism has been reported in pediatric patients presenting with slipped capital femoral epiphysis (SCFE), but never in patients with ectopic parathyroid adenoma. A 12-year-old boy with obesity and autism spectrum disorder presented with a limp and was found to have bilateral SCFE. Calcium was elevated to 12.3 mg/dL with parathyroid hormone (PTH) of 1191 pg/mL. Neck ultrasound revealed no parathyroid adenoma. He was discharged following bilateral surgical pinning with plans for outpatient workup. Repeat labs five days later demonstrated calcium had risen to 16.7 mg/dL. Tc-99m Sestamibi scintigraphy and CT scan revealed a 2.7 x 1.6 x 1.9 cm intrathymic mediastinal lesion. He underwent a thoracoscopic resection of the mass, and intraoperative PTH levels fell appropriately. Pathology revealed a parathyroid adenoma. Post-operatively, the patient developed hungry bone syndrome followed by normocalcemic secondary hyperparathyroidism which resolved with high-dose vitamin D supplementation. Primary hyperparathyroidism presenting as SCFE in a pediatric patient has been reported in thirteen previous cases. This is the first reported case of bilateral SCFE arising from an ectopic parathyroid adenoma. Thoracoscopic resection is a relatively new approach in pediatrics. Primary hyperparathyroidism can be associated with SCFE, especially bilateral, and should be considered in patients with traditional risk factors for SCFE. Pediatric patients with primary hyperparathyroidism and negative neck imaging should be further evaluated for ectopic parathyroid adenomas with nuclear medicine or cross-sectional imaging that includes the head, neck, and mediastinum. Thoracoscopic resection can be considered in pediatric patients with mediastinal ectopic parathyroid adenoma.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Primary Hyperparathyroidism From Ectopic Parathyroid Adenoma in a 12-Year-Old With Slipped Capital Femoral Epiphysis

Vitale

Shieh

Modi

et al. 2022

Journal of the Endocrine Society

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“…Serum calcium and PTH values represent useful diagnostic and follow-up tools; sometimes, PHP is unexpectedly identified due to a high calcium value at the moment when the patient is admitted for an apparently unrelated complaint, such as an infection [68]. Increased values of calcium require prompt medical intervention, including the decision of administering bisphosphonates and cinacalcet or, in refractory cases, emergency parathyroid tumor removal [66].…”

Section: Lab Findingsmentioning

confidence: 99%

Pediatric Neuroendocrine Neoplasia of the Parathyroid Glands: Delving into Primary Hyperparathyroidism

Carsote,

Stanciu,

Popa

et al. 2023

Biomedicines

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Our objective was to overview the most recent data on primary hyperparathyroidism (PHP) in children and teenagers from a multidisciplinary perspective. Methods: narrative review based on full-length, English-language papers (from PubMed, between January 2020 and July 2023). Results: 48 papers (14 studies of ≥10 subjects/study, and 34 case reports/series of <10 patients/study). Study-sample-based analysis: except for one case–control study, all of the studies were retrospective, representing both multicenter (n = 5) and single-center (n = 7) studies, and cohort sizes varied from small (N = 10 to 19), to medium-sized (N = 23 to 36) and large (N = 63 to 83); in total, the reviewed studies covered 493 individuals with PHP. Case reports/series (n = 34, N = 41): the mean ages studied varied from 10.2 to 14 years in case reports, and the mean age was 17 years in case series. No clear female predominance was identified, unlike that observed in the adult population. Concerning the assessments, there were four major types of endpoints: imaging data collection, such as ultrasound, 99mTc Sestamibi, or dual-phase computed tomography (CT); gene testing/familial syndrome identification; preoperative findings; and exposure to surgical outcome/preoperative drugs, like cinacalcet, over a 2.2-year median (plus two case reports of denosumab used as an off-label calcium-lowering agent). Single-gland cases (representing 85% of sporadic cases and 19% of genetic PHP cases) showed 100% sensitivity for neck ultrasounds, with 98% concordance with 99mTc Sestamibi, as well as a 91% sensitivity for dual-phase CT, with 25% of the lesions being ectopic parathyroids (mostly mediastinal intra-thymic). Case reports included another 9/41 patients with ectopic parathyroid adenomas, 3/41 with parathyroid carcinomas, and 8/41 subjects with brown tumors. Genetic PHP (which has a prevalence of 5–26.9%) mostly involved MEN1, followed by CDC73, CASR, RET, and CDKN1B, as well as one case of VHL. Symptomatic PHP: 70–100% of all cases. Asymptomatic PHP: 60% of genetic PHP cases. Renal involvement: 10.5% of a cohort with genetic PHP, 71% of sporadic PHP cases; 50% (in a cohort with a mean age of 16.7), 29% (in a cohort with a mean age of 15.2); 0% (in infancy) to 50–62% (in teenagers). Bone anomalies: 83% of the children in one study and 62% of those in two other studies. Gastrointestinal issues: 40% of one cohort, but the data are heterogeneous. Cure rate through parathyroidectomy: 97–98%. Recurrent PHP: 2% of sporadic PHP cases and 38% of familial PHP cases. Hungry bone syndrome: maximum rate of 34–40%. Case reports identified another 7/41 subjects with the same post-parathyroidectomy condition; a potential connection with ectopic presentation or brown tumors is suggested, but there are limited data. Minimally invasive thoracoscopic approaches for ectopic tumors seemed safe. The current level of statistical evidence on pediatric PHP qualifies our study- and case-sample-based analysis (n = 48, N = 534) as one of the largest of its kind. Awareness of PHP is the key factor to benefit our young patients.

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“…Ectopic parathyroid adenomas are even more rare and often present as a diagnostic challenge. Among 34 published case reports of ectopic parathyroid adenoma in pediatric patients up to the age of 18 years, 15 presented with an intrathymic parathyroid adenoma ( 4 ). Other locations of ectopic adenomas were in the mediastinum, within the carotid sheath, or retropharyngeal ( 4 ).…”

Section: Introductionmentioning

confidence: 99%

“…Among 34 published case reports of ectopic parathyroid adenoma in pediatric patients up to the age of 18 years, 15 presented with an intrathymic parathyroid adenoma ( 4 ). Other locations of ectopic adenomas were in the mediastinum, within the carotid sheath, or retropharyngeal ( 4 ). We present a 9-year-old girl with symptomatic primary hyperparathyroidism from an ectopic parathyroid adenoma located in the piriform sinus.…”

Section: Introductionmentioning

confidence: 99%

Case Report: Nine-year-old with parathyroid adenoma within the piriform sinus

et al. 2023

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We present the case of a 9-year-old girl who presented with symptomatic hypercalcemia from primary hyperparathyroidism (PHPT). Laboratory results revealed elevated serum calcium 12.1 mg/dl (ref: 9.1–10.4), elevated ionized calcium 6.8 (ref: 4.5–5.6) mg/dl, phosphorus 3.8 (ref: 3.3–5.1) mg/dl, 25-OH vitamin D 20.1 (30–100) ng/ml, and elevated intact PTH 70 (15-65) pg/ml, consistent with the diagnosis of PHPT. She had persistent hyperparathyroidism after bilateral neck exploration, left thyroid lobectomy, and transcervical thymectomy. Neither inferior gland was identified. No parathyroid tissue was seen on histology. Repeat preoperative imaging identified a 7-mm × 5-mm adenoma on 4DCT not seen on 99Tc-sestamibi parathyroid scan. The patient then underwent a successful redo parathyroidectomy with removal of a submucosal left parathyroid adenoma at the superior aspect of the thyroid cartilage in the piriform sinus. Her biochemical work-up remains consistent with surgical cure 6 months after surgery. Herein, we also review common locations for ectopic parathyroid adenomas.Clinical Trial RegistrationNCT04969926.

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Ectopic Parathyroid Adenoma in an 11-Year-Old Girl: Case Report and Literature Review

Cited by 6 publications

References 36 publications

Primary Hyperparathyroidism From Ectopic Parathyroid Adenoma in a 12-Year-Old With Slipped Capital Femoral Epiphysis

Primary Hyperparathyroidism From Ectopic Parathyroid Adenoma in a 12-Year-Old With Slipped Capital Femoral Epiphysis

Pediatric Neuroendocrine Neoplasia of the Parathyroid Glands: Delving into Primary Hyperparathyroidism

Case Report: Nine-year-old with parathyroid adenoma within the piriform sinus

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